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      Refractory Coats’ Disease of Adult Onset

      case-report

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          Abstract

          Purpose

          We present the case of an 18-year-old Caucasian male with a unilateral macular star and retinal vascular anomalies compatible with adult onset Coats’ disease.

          Methods

          Diagnosis was based on fundoscopic, fluorescein angiography and optical coherence tomography findings.

          Results

          The patient presented to our emergency department with complaints of low vision in his left eye (LE) detected 10 days before. The best-corrected visual acuity in the LE was 20/50. Fundoscopy of the LE evidenced a complete macular star. Optical coherence tomography showed increased retinal thickness, infiltration of the retinal wall, and detachment of the neuroepithelium. Angiography revealed no appreciable diffusion in the macula. Above the superior temporal (ST) arcade, anomalies in the retinal vasculature were found, with interruption of the peripheral vessels and vessels which were ‘sausage’-like. After 1 month, the LE vision evolved to hand movements. Laser photocoagulation was performed in the ST quadrant. Intravitreal injection of bevacizumab 1.25 mg/0.05 ml and photodynamic therapy were performed without any significant changes, progression of ST serous detachment of the neuroepithelium, and finally progression to macular fibrosis.

          Discussion

          Coats’ disease is usually diagnosed in childhood, but rare cases may occur in adults. Those cases usually have a more indolent course which was not observed in our patient. When there is macular involvement, prognosis is more guarded, despite treatment.

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          Most cited references12

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          Forms of retinal disease with massive exudation

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            Coats' disease diagnosed in adulthood.

            To investigate the manifestations and clinical course of Coats' disease diagnosed in adulthood and to describe the characteristics of patients who develop it. Retrospective observational case series. Thirteen eyes of 13 patients with Coats' disease diagnosed after age 35 years. All patients examined in the authors' referral practices for Coats' disease diagnosed after the age of 35 years were identified. Patients with a history of radiation exposure, intraocular inflammation, retinal vascular occlusion, or Coats' response were excluded. A review of their historical, clinical, and fluorescein angiographic features and demographics was performed. There were 13 patients with a mean age at diagnosis of 50 years (range, 36-79) and a mean follow-up of 5.8 years (range, 0-17). These patients manifested many findings typical of Coats' disease, including the unilateral nature of the disease (13/13 patients), male predominance (12/13), vascular telangiectasis, lipid exudation, macular edema, and areas of capillary nonperfusion with adjacent webs of filigreelike capillaries. Disease was limited to < or =6 clock hours in 10 of 13 patients (76.9%), did not generally involve the retinal vasculature anterior to the equator, and showed limited potential to expand during the follow-up period. A localized hemorrhage was noted in 10 of 13 patients (76.9%), almost always near larger aneurysmal dilatations. Limited exudative detachment of the retina was seen in 4 eyes, 3 of which responded to localized laser photocoagulation. One patient had subretinal fluid seen on ocular coherence tomography that did not require treatment. On average, patients lost 2.1 lines of visual acuity during the follow-up period; 6 patients had a decline in vision, 2 patients had improved vision, and 3 patients had stable vision. No patient had end-stage findings of Coats' disease, such as iris neovascularization or total exudative detachment. Coats' disease can first be diagnosed in adulthood with retinal vascular abnormalities similar to those seen in younger patients. There are a number of important differences in disease manifestation in adults, including limited area of involvement, slower apparent progression of disease, and hemorrhage near larger vascular dilatations.
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              Resolution of Severe Macular Edema in Adult Coats' Disease with Intravitreal Triamcinolone and Bevacizumab Injection

              A 47 year old male patient visited our hospital with the chief complaint of deterioration of the visual acuity in the left eye. The fundus examination revealed thick hard exudates, multiple aneurysms and telangiectasias of the retinal vessels in the posterior pole. Fluorescein angiography demonstrated massive leakage over an area of the aneurysms. Optical coherence tomography (Stratus OCT; Zeiss-Humphrey, Dubin, CA) revealed diffuse and marked thickening of the retina. Laser photocoagulation was performed under the diagnosis of Coats' disease. However, the treatment could not be performed satisfactorily. On the first and 6th weeks, an intravitreal injection of bevacizumab and triamcinolone acetonide was administered, and laser photocoagulation was again attempted. The effectiveness of eachagent on retinal edema was evaluated at the follow-up performed at 1, 2, 5, 7, 10 weeks and 6 months after the injection. At one week after the intravitreal bevacizumab injection, there was no improvement. An intravitreal injection of triamcinolone acetonide was performed 6 weeks after the initial diagnosis,which resulted in a reduction in the thickness of the macular edema. Therefore, laser photocoagulation was performed sufficiently on telangiectasias. The follow-up at 6 months showed a relative increase in the macular edema, but there was reduced leakage from the telangiectasias compared with the previous angiograph.
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                Author and article information

                Journal
                Case Report Ophthalmol
                Case Report Ophthalmol
                COP
                Case Reports in Ophthalmology
                S. Karger AG (Allschwilerstrasse 10, P.O. Box · Postfach · Case postale, CH–4009, Basel, Switzerland · Schweiz · Suisse, Phone: +41 61 306 11 11, Fax: +41 61 306 12 34, karger@karger.ch )
                1663-2699
                Jan-Apr 2012
                26 March 2012
                26 March 2012
                : 3
                : 1
                : 118-122
                Affiliations
                Department of Ophthalmology, Hospital Santo André, Leiria, Portugal
                Author notes
                *Diana Garcia Beselga, Department of Ophthalmology, Santo Andrés Hospital, Olhalvas St., Pousos, PT-2410-197 Leiria (Portugal), E-Mail dianabeselga@ 123456hotmail.com
                Article
                cop-0003-0118
                10.1159/000337642
                3339686
                22548045
                42f0b9e2-d3e2-4f71-9428-d50987a08295
                Copyright © 2012 by S. Karger AG, Basel

                This is an Open Access article distributed under the terms of the Creative Commons Attribution-Noncommercial-No-Derivative-Works License ( http://creativecommons.org/licenses/by-nc-nd/3.0/). Users may download, print and share this work on the Internet for noncommercial purposes only, provided the original work is properly cited, and a link to the original work on http://www.karger.com and the terms of this license are included in any shared versions.

                History
                Page count
                Figures: 4, References: 13, Pages: 5
                Categories
                Published: March, 2012

                Ophthalmology & Optometry
                macular star,laser photocoagulation,intravitreal bevacizumab,photodynamic therapy,coats’ disease,adult onset

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